Cui Dong, Hu Yu-Hui, Tang Gen, Shen Dan, Chen Li, Liao Jian-Xiang, Chen Shu-Li
Department of Inherited Metabolic Diseases, Shenzhen Children's Hospital Affiliated to Medical College of Shantou University, Shenzhen 518038, China.
Zhongguo Dang Dai Er Ke Za Zhi. 2019 Apr;21(4):375-380. doi: 10.7499/j.issn.1008-8830.2019.04.014.
Lysinuric protein intolerance (LPI) is an autosomal recessive disorder caused by SLC7A7 gene mutation and often involves severe lesions in multiple systems. Lung involvement is frequently seen in children with LPI and such children tend to have a poor prognosis. This article summarizes the clinical manifestations and gene mutation characteristics of three children diagnosed with LPI by SLC7A7 gene analysis. All three children had the manifestations of aversion to protein-rich food after weaning, delayed development, anemia, hepatosplenomegaly, and osteoporosis, as well as an increase in orotic acid in urine. In addition, interstitial pneumonia and diffuse pulmonary interstitial lesions were observed in two children. SLC7A7 gene detection showed three pathogenic mutations in these children, namely c.1387delG(p.V463CfsX56), c.1215G>A(p.W405X) and homozygous c.625+1G>A. After a definite diagnosis was made, all three children were given a low-protein diet and oral administration of citrulline [100 mg/(kg.d)], iron protein succinylate [4 mg/(kg.d)], calcium and zinc gluconates oral solution (10 mL/day) and vitamin D (400 IU/day). In addition, patient 3 was given prednisone acetate (5 mg/day). The children had varying degrees of improvement in symptoms and signs. It is hard to distinguish LPI from urea cycle disorder due to the features of amino acid and organic acid metabolism in LPI, and SLC7A7 gene analysis is the basis for a definite diagnosis of LPI.
赖氨酸尿性蛋白不耐受症(LPI)是一种由SLC7A7基因突变引起的常染色体隐性疾病,常累及多个系统的严重病变。LPI患儿肺部受累较为常见,且这类患儿预后往往较差。本文总结了3例经SLC7A7基因分析确诊为LPI患儿的临床表现及基因突变特征。3例患儿均有断奶后厌恶富含蛋白质食物、发育迟缓、贫血、肝脾肿大、骨质疏松以及尿中乳清酸升高的表现。此外,2例患儿观察到间质性肺炎和弥漫性肺间质病变。SLC7A7基因检测显示这些患儿存在3种致病突变,即c.1387delG(p.V463CfsX56)、c.1215G>A(p.W405X)和纯合子c.625+1G>A。确诊后,3例患儿均给予低蛋白饮食,并口服瓜氨酸[100mg/(kg·d)]、琥珀酸亚铁蛋白[4mg/(kg·d)]、葡萄糖酸钙锌口服溶液(10ml/天)和维生素D(400IU/天)。此外,患儿3给予醋酸泼尼松(5mg/天)。患儿症状和体征均有不同程度改善。由于LPI中氨基酸和有机酸代谢的特点,LPI很难与尿素循环障碍相鉴别,而SLC7A7基因分析是确诊LPI的依据。